Case Report Pleomorphic Adenoma: Case Report and Review of Literature N. Balan, M. Sudhaa Mani, S. Yasmeen Ahamed, D. A. Divya

Department of Oral Medicine Pleomorphic adenoma, the most common salivary gland tumor, consists of and Radiology, Vivekanandha epithelial and mesenchymal components. Its morphologic complexity results Dental College for Women, Namakkal, Tamil Nadu, India from differentiation of tumor cells into fibrous, hyalinized, myxoid, chondroid, and osseous areas. The diagnosis is made by the clinical and histopathological Abstract examination. The occurrence of pleomorphic adenoma in upper lip is very rare. Kroll and Hick reviewed 4042 cases of pleomorphic adenomas of the salivary glands. Of these, 445 originated in the minor salivary glands, of which only 16.9% were located in the upper lip and 2.9% in the lower lip. Pleomorphic adenomas of the minor salivary glands generally present as painless, submucosal swellings. We report a case of pleomorphic adenoma which presents as a swelling of upper lip for a 72‑year‑old female patient.

Keywords: Minor salivary gland, mixed tumor, upper lip

Introduction 3 cm × 2 cm in size. It extended superiorly 3 cm from he term pleomorphic adenoma was suggested by inner canthus of the eye, inferiorly to vermillion border T Willis. In earlier years, it was referred by names of the upper lip, medially from infranasal depression such as enclavoma, branchioma, endothelioma, and laterally to nasolabial fold. There was deviation of the enchondroma.[1] It is the commonest of the salivary nasal septum to the right side, elevation of left ala of gland tumors, accounting for 50%–70% of cases of nose and obliteration of nasolabial fold. The surface parotid tumors, 40%–60% submandibular tumors, and appeared smooth with no secondary changes. On 10% minor salivary gland tumors, with palate (60%) palpation, the swelling had no local rise in temperature, being the most common followed by upper lip (20% nontender, firm in consistency, and freely mobile. No of cases).[2] The most commonly affected age group pulsations were felt [Figure 1]. Bilateral submandibular are in the fourth, fifth, and sixth decades; 60% of them lymph nodes were palpable, firm in consistency and are female.[3] It has been suggested that 25% of benign mobile. mixed tumors undergo malignant transformation. On intraoral examination, a well‑defined swelling was present on the left maxillary labial mucosa, ovoid, Case Report 3 cm × 2 cm in size, surface was smooth and mucosa A 72‑year‑old female patient reported to the Department over the swelling appeared pale pink, extends anteriorly of Oral Medicine and Radiology with the complaint of from maxillary labial frenum, posteriorly 1 cm away from swelling in the left middle‑third of face for the past buccal frenum, superiorly 0.5 cm from vermillion border 1 year. History revealed that the swelling was gradual of upper lip, inferiorly to attached gingiva. There were no in onset and slowly increased in size for the past 1 year. secondary changes such as sinus opening or pus discharge The swelling was asymptomatic. Patient reported of the or ulceration. On palpation, it was nontender, firm in nasal stiffness of the left nostril and associated difficulty in breathing. There was no history of trauma, loss of Address for correspondence: Dr. M. Sudhaa Mani, appetite, and loss of weight. Department of Oral Medicine and Radiology, Vivekanandha Dental College for Women, Tiruchengode, Namakkal, On extraoral examination, a well‑defined swelling Tamil Nadu, India. was present on left middle‑third of the face, oval, E‑mail: [email protected]

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How to cite this article: Balan N, Mani MS, Ahamed SY, Divya DA. DOI: 10.4103/jiadsr.jiadsr_17_17 Pleomorphic adenoma: Case report and review of literature. J Indian Acad Dent Spec Res 2017;4:61-4.

© 2017 Journal of Indian Academy of Dental Specialist Researchers | Published by Wolters Kluwer - Medknow 61 Balan, et al.: Pleomorphic adenoma consistency, compressible, and mobile. No pulsations pleomorphic adenoma [Figure 5a and b]. Follow up was were felt [Figure 2]. Based on the history and clinical done after 1 month and patient was free of symptoms findings, a provisional diagnosis of benign salivary gland [Figure 6]. tumor was considered probably pleomorphic adenoma. The differential diagnosis was fibrosed mucocele, Discussion peripheral fibroma, myxoma, and lipoma. Fine‑needle Pleomorphic adenoma is a benign neoplasm which aspiration cytology was negative. Intraoral periapical consists of cells exhibiting the ability to differentiate radiograph and the OPG revealed no bony changes. CT into both epithelial (ductal and nonductal) cells and report revealed a hypodense area of size 3 cm × 2 cm mesenchymal (chondroid, myxoid, and osseous) cells. seen in relation to upper lip with mild scalloping of the The most common site among the major salivary gland is underlying maxilla [Figure 3a and b]. Excisional biopsy parotid (approximately 75%) submandibular gland (around was done under GA and the specimen was submitted 5%–10%) and the minor salivary gland (approximately for histopathological examination [Figure 4a and b] 10%). In a study conducted in Indian population during Microscopic analysis of the surgical specimen revealed 5 years duration, out of 5515 tumors involving various an encapsulated mass with nests of epithelial cells in organs, 53 were salivary gland tumors accounting for a background of myxoid stroma. These epithelial cells 0.96% of all neoplasms.[4] Most cases of pleomorphic exhibit large nuclei with eosinophilic cytoplasm and adenomas (70%) show cytogenetic aberrations.[5] The indistinct cell borders. The surrounding myxoid stroma mucin 1 gene has been associated with malignant reveals widely separated angular shaped cells with transformation of this tumor.[6] The literature suggests that numerous neurovascular bundles. The final diagnosis was the simian virus (SV40) may play a causative role in the

Figure 1: Extraoral photograph Figure 2: Intraoral preoperative view

a

a b Figure 3: (a) Axial computed tomography scan image showing hypodense area in left premaxillary region. (b) Coronal computed tomography scan b image showing hypodense area in left premaxillary region Figure 4: (a) Excision of lesion. (b) Measurement of the lesion

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a b Figure 5: (a) Histopathologic view ×40. (b) Histopathologic view ×10 development of pleomorphic adenoma. Putative risk factors include cigarette smoking, genetic predisposition, viral infections, rubber manufacturing, plumbing, some types of woodworking, as well as asbestos mining, exposure to nickel compounds, and cellular phone use.[7] The only Figure 6: Postoperative view after 1 month well‑established risk factor is ionizing radiation. Atomic bomb survivors and cancer patients treated by radiation Prognosis is excellent as the tumor is benign, and present with a substantially higher risk of developing salivary recurrence is not being noted. gland tumors. The typical presentation is asymptomatic, slow growing, painless, firm mass, nontender, and tends Declaration of patient consent to be mobile when small but fixed to surround tissue with The authors certify that they have obtained all appropriate advanced growth.[8] In our case, the tumor presents as patient consent forms. In the form the patient(s) has/have asymptomatic slow growing swelling which is nontender given his/her/their consent for his/her/their images and and firm in consistency and mobile. The extent and depth other clinical information to be reported in the journal. of the lesion cannot be accurately seen in conventional The patients understand that their names and initials will radiographs, henceforth the 3‑d imaging modalities such as not be published and due efforts will be made to conceal computed tomography (CT) scan and magnetic resonance their identity, but anonymity cannot be guaranteed. imaging are considered to be gold standard in imaging Financial support and sponsorship such lesions. In our case, no bony changes seen in intraoral Nil. periapical and panoramic radiograph. On CT images, a hypodense area of size 3 cm × 2 cm seen in relation to Conflicts of interest upper lip with mild scalloping of the underlying maxilla. There are no conflicts of interest. Radical surgical excision is the cornerstone treatment of salivary gland tumors.[9] If complete resection cannot References be achieved, adjuvant radiotherapy should be added to 1. Sunil S, Gopakumar D. Pleomorphic adenoma. A case report and improve local control.[10] The differential diagnosis of review of literature. Int. J. Odontostomat 2013;7:171-4. 2. Pardhe ND, Vijay P, Singhal I, Shah G, Pleomorphic adenoma of asymptomatic nodules involving minor salivary gland are upper lip: Case report. IJOCR 2015;3:100-3. [11] neurofibroma, lipoma, and rhabdomyosarcoma. In the 3. Rahnama M, Orzędała‑Koszel U, Czupkałło L, Lobacz M. study by Neville et al., 92% of the upper lip tumors were Pleomorphic adenoma of the palate: A case report and review of monomorphic adenoma (canalicular adenoma and basal the literature. Contemp Oncol (Pozn) 2013;17:103‑6. cell adenoma) and pleomorphic adenoma, whereas sporadic 4. Srivani N, Srujana S, Shahista S, Shravan Kumar O. Spectrum of salivary gland tumors – A five year study. IAIM cases of adenoid cystic carcinoma, acinic carcinoma, and 2016;3:132‑6. adenocarcinoma constitute the remainder. Malignant tumors 5. Martins C, Fonseca I, Roque L, Pereira T, Ribeiro C, Bullerdiek J, tend to predominate in the lower lip.[12] Most recurrences et al. PLAG1 gene alterations in salivary gland pleomorphic are due to inadequate surgical techniques such as simple adenoma and carcinoma ex‑pleomorphic adenoma: A combined enucleation leaving behind microscopic pseudopod‑like study using chromosome banding, in situ hybridization and immunocytochemistry. Mod Pathol 2005;18:1048‑55. [13] extensions. 6. Namboodiripad PC. A review: Immunological markers for malignant salivary gland tumors. J Oral Biol Craniofac Res Conclusion 2014;4:127‑34. Pleomorphic adenoma generally does not recur after 7. Sadetzki S, Chetrit A, Jarus‑Hakak A, Cardis E, Deutch Y, Duvdevani S, et al. Cellular phone use and risk of benign and adequate surgical excision. Ultimately, complete surgical malignant parotid gland tumors – a nationwide case‑control excision will provide the definitive diagnosis and study. Am J Epidemiol 2008;167:457‑67. treatment for this noteworthy salivary gland neoplasm. 8. Gajjar HK, Shah CK, Shah FR. Pleomorphic adenoma (minor

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salivary gland) of upper lip. J Med Sci July 2013;2:82-4. 11. Kataria SP, Tanwar P, Sethi D, Garg M. Pleomorphic adenoma of 9. Witt RL, Eisele DW, Morton RP, Nicolai P, Poorten VV, the upper lip. J Cutan Aesthet Surg 2011;4:217-9. Zbären P, et al. Etiology and management of recurrent parotid 12. Neville BW, Damm DD, Weir JC, et al. Labial salivary gland pleomorphic adenoma. Laryngoscope 2015;125:888‑93. tumors. Cancer 1988;61:2113-6 10. Debnath SC, Saikia AK, Debnath A, Pleomorphic adenoma of 13. Feinmesser R, Gay I. Pleomorphic adenoma of the hard palate. the palate. J Maxillofac Oral Surg 2010;9:420-3. J Laryngol Otol 1983;97:1169‑71.

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